Ultrasonographic diagnosis of congenital membranous jejunal stenosis and gastric

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Qinghua et al. Journal of Medical Case Reports (2015) 9:162
DOI 10.1186/s13256-015-0644-2
JOURNAL OF MEDICAL
CASE REPORTS
CASE REPORT
Open Access
Ultrasonographic diagnosis of congenital
membranous jejunal stenosis and gastric
duplication cyst in a newborn: a case report
Liu Qinghua1*, Wu Shoucai2, Liu Xiaofang1, Zhang Xincun1 and Miao Lili1
Abstract
Introduction: Congenital jejunal stenosis and gastric duplication cysts are very rare congenital anomalies of the
gastrointestinal tract in the newborn. We present a case of congenital membranous jejunal stenosis associated with
gastric duplication cysts, which was diagnosed by ultrasonography. To the best of our knowledge, this is the
first report of ultrasonographic diagnosis of congenital membranous jejunal stenosis associated with a gastric
duplication cyst in a newborn.
Case presentation: A 1-month-old Chinese baby girl presented with projectile vomiting and hyperpyrexia for 3 days.
An upper gastrointestinal contrast study showed incomplete duodenal obstruction; however, ultrasonography revealed
congenital membranous jejunal stenosis associated with a gastric duplication cyst. After surgical excision of the jejunal
membrane and gastric duplication cyst, she recovered well with no bilious vomiting at the 1-year follow-up.
Conclusion: Ultrasonography is a useful tool for the evaluation of membranous jejunal stenosis and the identification of
small, asymptomatic gastrointestinal duplication cysts.
Keywords: Congenital membranous jejunal stenosis, Gastric duplication cyst, Newborn, Ultrasonography
Introduction
Congenital jejunal stenosis and gastric duplication cysts are
very rare congenital anomalies of the gastrointestinal (GI)
tract in the newborn; the prevalence rate of jejunoileal
atresia and stenosis has been reported to be between 1 in
330 and 0.9 in 10,000 live births, and stenosis occurs in 7 %
of cases [1]. The incidence of duplication cysts of the alimentary system is approximately 1 in 4500 live births [2],
and gastric duplication cysts represent only 4 to 5 % of
these cases [3, 4]. We present a case of congenital membranous jejunal stenosis associated with gastric duplication
cysts, which was diagnosed by ultrasonography. The patient
underwent surgery and recovered well. To the best of our
knowledge, this is the first report of ultrasonographic diagnosis of congenital membranous jejunal stenosis associated
with a gastric duplication cyst in a newborn.
* Correspondence: xr6547@163.com
1
Department of Ultrasound, Jinan Children’s Hospital, Jinan 250022, China
Full list of author information is available at the end of the article
Case presentation
A 1-month-old Chinese baby girl presented to our emergency department with vomiting and high-grade fever,
which had occurred for 3 days. The fever was not associated with rigors and chills. She presented with a history of
intermittent projectile and bilious vomiting after breastfeeding. Vomiting occurred approximately five times each day,
at a volume of 10mL at each instance. A general physical
examination revealed a temperature of 39 °C, pulse rate of
100/minute, and respiratory rate of 30/minute. On abdominal examination, no abdominal distention, regional tenderness, or rebound was observed, and no masses or viscera
could be palpated. Bowel sounds were audible, and other
physical examination results were normal. An upper GI
contrast study in another hospital showed incomplete duodenal obstruction (Fig. 1). However, to clarify the cause of
intestinal obstruction, she subsequently underwent
ultrasonographic examination after hospitalization. Ultrasonography showed continuous opening of the pylorus
and significant dilatation of her duodenum; her upper
jejunum in the left abdomen was approximately 4cm in
diameter, and we observed frequent reverse peristalsis.
© 2015 Qinghua et al. This is an Open Access article distributed under the terms of the Creative Commons Attribution License
(http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium,
provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://
creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
Qinghua et al. Journal of Medical Case Reports (2015) 9:162
Page 2 of 5
Fig. 1 Upper gastrointestinal contrast study showing marked dilatation of the duodenum
An intraluminal membrane was visible in the dilated
distal jejunum site (Fig. 2), and the “billow” in the proximal jejunum could be seen to intermittently move in
and out of a pinhole in the mucosal diaphragm. The
distal intestine was deflated, and there was an absence
of gas; by ultrasonography, a diagnosis of congenital
membranous jejunal stenosis was considered. Furthermore, an ultrasound showed a cystic structure sized
2.0 × 1.5cm in the greater curvature in the left hypochondrium (Fig. 3). It was firmly attached at the gastric
wall, and the cyst walls consisted of an inner echogenic
rim and an outer echogenic rim area. The central area
was hypoechoic. A gastric duplication cyst was considered the most likely diagnosis, and a laparotomy was
planned after initial stabilization. During surgery, jejunal
stenosis situated approximately 15cm from the ligament
of Treitz and a diaphragm with a pinhole in the intestinal
cavity was found and resected. In addition, surgical excision of the cyst located on the greater curvature of the
stomach was performed. A histopathological examination
revealed that both sides of the resected intestinal membrane were covered with intestinal mucosa (Fig. 4) and the
structure of the gastric mucosa and smooth muscles in
the wall of the cyst, confirming the diagnosis of
Fig. 2 Ultrasonography showing dilatation of the duodenum and proximal jejunum. A membrane (arrow) and a pinhole (small arrowhead) are
seen in the jejunal membrane
Qinghua et al. Journal of Medical Case Reports (2015) 9:162
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Fig. 3 Ultrasonographic image of the gastric duplication cyst. It was 1.5 × 1.4cm and intimately attached at the greater curvature of the stomach
membranous jejunal stenosis and gastric duplication cyst
(Fig. 5). Postoperative recovery was uneventful, and she
was discharged on the ninth day following surgery.
She recovered well with no bilious vomiting at the
1-year follow-up.
Discussion
In general, the symptoms of patients with congenital
anomalies of the alimentary tract present early after
birth with recurrent, progressive vomiting. Bilious
vomiting indicates obstruction distal to the ampulla
of Vater [5], which requires an immediate and correct
diagnosis and surgical correction to avoid serious
complications. Normally, within 3 hours of birth, the
entire small bowel contains gas, whereas gas is observed in the sigmoid colon 8 to 9 hours after birth
[6]. In some patients with congenital anomalies of the
GI tract causing obstructions, the proximal bowel
with lesions exhibits dilatation and effusion, and the
distal intestinal cavity has little or no gas, thereby
creating favorable conditions for ultrasonic detection
of the lesions. Furthermore, ultrasonography of the
GI tract is noninvasive, simple, radiation-free, and
fairly accurate for the diagnosis of GI obstruction.
Therefore, ultrasonography plays a vital role in the
evaluation of patients with congenital anomalies of
the GI tract. Ultrasonography has been used as an alternative to barium studies in diagnosing certain congenital anomalies of the GI tract, including annular
pancreas, hypertrophic pyloric stenosis, enteric duplication cysts, malrotation, and midgut volvulus [5].
However, the use of ultrasonography for the detection and
diagnosis of membranous jejunal stenosis has not been
previously reported in the literature. A previous study
reported that jejunoileal atresia and stenosis are difficult
to evaluate [7]. In the present case, ultrasonography revealed the mucosal diaphragm with a pinhole in the jejunum, the duodenum and proximal jejunum loops
significantly dilated with fluid, and the distal small intestine deflated with little gas. Based on this case, ultrasonography may have greater importance in the evaluation of
membranous jejunal stenosis, as it enables the distinction
from atresia and provides excellent anatomic details for
correct diagnosis and treatment planning.
Duplication cysts are rare congenital anomalies that can
occur in any part of the GI tract, although the most common site is the ileum [3]. Gastric duplication cysts represent only 4 to 5% of duplication cysts and are located
along the greater curvature of the stomach, usually in the
antrum [3, 4]. Possible causes of gastric duplication cysts
Fig. 4 Histopathological examination showing both sides of the
resected intestinal membrane covered with intestinal mucosa
Qinghua et al. Journal of Medical Case Reports (2015) 9:162
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Fig. 5 Histological examination of the resected cyst. Gastric mucosa and smooth muscles are seen in the wall of the cyst
include errors in recanalization of the alimentary tract,
McLetchie’s theory of traction diverticulum formation
caused by endo-ectodermal adhesions, split notochord,
and partial twinning [8, 9]. On clinical examination, gastric duplication cysts can present with vague abdominal
pain, vomiting, and occasionally a palpable abdominal
mass; however, presentation is usually asymptomatic and
therefore difficult to diagnose [10]. In the present case, a
gastric duplication cyst was incidentally diagnosed by
abdominal ultrasonography. Ultrasonography showed a
small cystic lesion on the greater curvature of the stomach
in the left hypochondrium. Characteristic ultrasonographic features of a GI duplication cyst are a hypoechoic
muscular rim surrounding an echogenic mucosa; these
features are specific to gastric duplication cysts and are
not seen with other cystic lesions [11]. However, in the
present case, an upper GI tract contrast study was performed, with no evidence of mass lesions. This case report
illustrates the important role of ultrasonography in detecting small, unsuspected duplication cysts.
accompanying images. A copy of the written consent is
available for review by the Editor-in-Chief of this journal.
Abbreviation
GI: Gastrointestinal.
Competing interests
The authors declare that they have no competing interests.
Authors’ contributions
LQ performed the ultrasonographic examination of the case, and was a
major contributor in writing the manuscript. WS obtained the relevant
literature. LX collected the patient’s clinical data. ZX and ML translated and
proofread the manuscript in English. All authors read and approved the
manuscript.
Acknowledgments
We are very grateful to our supervisor, Editage, and Dr Zheng Wenhui, who
provided valuable guidance at every stage of the writing of this report.
Author details
1
Department of Ultrasound, Jinan Children’s Hospital, Jinan 250022, China.
2
Department of Ultrasound, Qilu Hospital, Shandong University, Jinan
250012, China.
Received: 24 February 2015 Accepted: 29 June 2015
Conclusions
Using ultrasonography, we successfully diagnosed congenital membranous jejunal stenosis associated with a
gastric duplication cyst in a newborn. This case emphasizes that ultrasonography in a baby with congenital
jejunal stenosis was effective as a diagnostic modality. In
addition, ultrasonography can be helpful for the accurate
identification of small and asymptomatic GI duplication
cysts. Ultrasonography is superior to radiography for revealing the cause of some intestinal obstructions, suggesting that babies with bilious vomiting should have a
routine ultrasound for diagnosis.
Consent
Written informed consent was obtained from the patient’s
legal guardian for publication of this case report and
References
1. Stollman TH, de Blaauw I, Wijnen MH, van der Staak FH, Rieu PN, Draaisma
JM, et al. Decreased mortality but increased morbidity in neonates with
jejunoileal atresia; a study of 114 cases over a 34-year period. J Pediatr Surg.
2009;44:217–21.
2. Potter EL. Pathology of the fetus and newborn. 2nd ed. Chicago: Year Book
Medical Publishers; 1961.
3. Faerber EN, Balsara R, Vinocur CD, de Chadarevian JP. Gastric duplication
with hemoptysis: CT findings. AJR Am J Roentgenol. 1993;161:1245–6.
4. Sieunarine K, Manmohansingh E. Gastric duplication cyst presenting as an
acute abdomen in a child. J Pediatr Surg. 1989;24:1152.
5. Gupta AK, Guglani B. Imaging of congenital anomalies of the
gastrointestinal tract. Indian J Pediatr. 2005;72:403–14.
6. Berrocal T, Lamas M, Gutieérrez J, Torres I, Prieto C, del Hoyo ML. Congenital
anomalies of the small intestine, colon, and rectum. Radiographics.
1999;19:1219–36.
7. Sunada K, Yamamoto H, Kita H, Yano T, Sato H, Hayashi Y, et al. Clinical
outcomes of enteroscopy using the double-balloon method for strictures of
the small intestine. World J Gastroenterol. 2005;11:1087–9.
Qinghua et al. Journal of Medical Case Reports (2015) 9:162
Page 5 of 5
8.
Bonacci JL, Schlatter MG. Gastric duplication cyst: a unique presentation. J
Pediatr Surg. 2008;43:1203–5.
9. Shinde T, Lindner J, Silverman J, Agrawal R, Dhawan M. Gastric-duplication
cyst with an aberrant pancreatic-ductal system: an unusual cause of
recurrent abdominal pain. Gastrointest Endosc. 2009;69:377–9.
10. Prinsloo H, Loveland J, Grieve A, Andronikou S, Valli OM. Gastric duplication
cysts as a rare cause of haematemesis: diagnostic challenges in two
children. Pediatr Surg Int. 2011;27:1127–30.
11. Barr LL, Hayden Jr CK, Stransberry SD, Swischuk LE. Enteric duplication cysts
in children: are their ultrasonographic wall characteristics diagnostic? Pediatr
Radiol. 1990;20:326–8.
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